Introduction. Antiphospholipid syndrome may be associated with stroke. Case report. We report a case of a symptomatic occlusion of the innominate artery in a 37- year- old patient, who developed primary anti- phospholipid syndrome. This association, to our knowledge, has not been reported in the literature. Occlusion of the innominate artery was treated by angioplastystenting. Effective oral anticoagulation was given to prevent a new thrombotic event. Conclusion. Systematic search for antiphospholipid antibodies is needed as part of the etiology work- up in young stroke victims. We report a case of a symptomatic occlusion of the innominate artery in a 37-year-old patient, who developed primary anti-phospholipid syndrome. This association, to our knowledge, has not been reported in the literature. Occlusion of the innominate artery was treated by angioplastystenting. Effective oral anticoagulation was given to prevent a new thrombotic event. Conclusion. Systematic search for antiphospholipid antibodies is needed as part of the etiology work-up in young stroke victims .